{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T14:17:26Z","timestamp":1784557046092,"version":"3.55.0"},"reference-count":12,"publisher":"Bioscientifica","license":[{"start":{"date-parts":[[2022,12,1]],"date-time":"2022-12-01T00:00:00Z","timestamp":1669852800000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["edm.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2022,12,1]]},"abstract":"<jats:sec>\n                    <jats:title>Summary<\/jats:title>\n                    <jats:p>\n                      Tumor-induced osteomalacia (TIO) is a rare form of osteomalacia caused by fibroblast growth factor-23 (FGF23)-secreting tumors. Most of these tumors are phosphaturic mesenchymal tumors (PMTs) typically involving soft tissue in the extremities and bone of the appendicular skeleton and cranium. We report the case of a 60-year-old woman with about 3 years of persistent bone pain and multiple fractures, initially diagnosed as osteoporosis, who was found to have hypophosphatemia with low 1,25-dihydroxyvitamin D and elevated alkaline phosphatase and inappropriately normal FGF23 consistent with TIO. Her symptoms improved with phosphate supplementation, vitamin D and calcitriol.\n                      <jats:sup>68<\/jats:sup>\n                      Ga-DOTATATE imaging revealed a T12 vertebral body lesion confirmed on biopsy to be a PMT. She underwent resection of the PMT with resolution of TIO and increased bone density. This rare case of TIO secondary to a PMT of the thoracic spine highlights some of the common features of PMT-associated TIO and draws attention to PMT-associated TIO as a possible cause of unexplained persistent bone pain, a disease entity that often goes undiagnosed and untreated for years.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Learning points<\/jats:title>\n                    <jats:list list-type=\"bullet\">\n                      <jats:list-item>\n                        <jats:p>Tumor-induced osteomalacia (TIO) is typically caused by phosphaturic mesenchymal tumors (PMTs) that are usually found in the soft tissue of the extremities and bone of the appendicular skeleton\/cranium and rarely in the spine.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>TIO may be misdiagnosed as osteoporosis or spondyloarthritis, and the correct diagnosis is often delayed for years. However, osteoporosis, in the absence of fracture, is not associated with bone pain.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>The hallmark of TIO is hypophosphatemia with inappropriately normal or low 1,25-dihydroxyvitamin D and elevated or inappropriately normal fibroblast growth factor-23 (FGF23) levels.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>In patients with unexplained persistent bone pain, a serum phosphate should be measured. Consider PMT-associated TIO as a potential cause of unexplained persistent bone pain and hypophosphatemia.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>\n                          PMTs express somatostatin receptors and may be identified with\n                          <jats:sup>68<\/jats:sup>\n                          Ga-DOTATATE imaging.\n                        <\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>Complete surgical resection is the preferred treatment for spinal PMTs associated with TIO.<\/jats:p>\n                      <\/jats:list-item>\n                    <\/jats:list>\n                  <\/jats:sec>","DOI":"10.1530\/edm-22-0344","type":"journal-article","created":{"date-parts":[[2022,12,7]],"date-time":"2022-12-07T07:01:15Z","timestamp":1670396475000},"update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-2","source":"Crossref","is-referenced-by-count":3,"title":["Tumor-induced osteomalacia treated with T12 tumor resection"],"prefix":"10.1530","volume":"2022","author":[{"given":"Alyssa J","family":"Mancini","sequence":"first","affiliation":[{"name":"Harvard Medical School, Boston, MA, USA Hospital Medicine Unit, Department of Medicine, Massachusetts General Hospital, Boston, Massachusetts, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amin","family":"Sabet","sequence":"additional","affiliation":[{"name":"Boston University School of Medicine, Boston, Massachusetts Division of Endocrinology, Department of Medicine, St. Elizabeth\u2019s Medical Center, Boston, Massachusetts, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gunnlaugur Petur","family":"Nielsen","sequence":"additional","affiliation":[{"name":"Department of Pathology, Massachusetts General Hospital, Boston, Massachusetts, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J Anthony","family":"Parker","sequence":"additional","affiliation":[{"name":"Department of Radiology, Beth Israel Deaconess Medical Center, Boston, Massachusetts, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joseph H","family":"Schwab","sequence":"additional","affiliation":[{"name":"Department of Orthopedic Surgery, Massachusetts General Hospital, Boston, Massachusetts, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ashley","family":"Ward","sequence":"additional","affiliation":[{"name":"Department of Pathology, Massachusetts General Hospital, Boston, Massachusetts, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jim S","family":"Wu","sequence":"additional","affiliation":[{"name":"Department of Radiology, Beth Israel Deaconess Medical Center, Boston, Massachusetts, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7276-573X","authenticated-orcid":true,"given":"Alan O","family":"Malabanan","sequence":"additional","affiliation":[{"name":"Boston University School of Medicine, Boston, MA Section of Endocrinology, Diabetes and Nutrition, Boston Medical Center, Boston, Massachusetts, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"416","reference":[{"key":"bib1","series-title":"Modern Pathology","doi-asserted-by":"publisher","first-page":"189","DOI":"10.1038\/s41379-018-0100-0","article-title":"Phosphaturic mesenchymal tumor with an admixture of epithelial and mesenchymal elements in the jaws: clinicopathological and immunohistochemical analysis of 22 cases with literature review","volume":"32","author":"Wu","year":"2019","unstructured":"Wu HBui MMZhou LLi DZhang HZhong D. Phosphaturic mesenchymal tumor with an admixture of epithelial and mesenchymal elements in the jaws: clinicopathological and immunohistochemical analysis of 22 cases with literature review. Modern Pathology201932189\u2013204. (https:\/\/doi.org\/10.1038\/s41379-018-0100-0)10.1038\/s41379-018-0100-0"},{"key":"bib2","series-title":"Seminars in Diagnostic Pathology","doi-asserted-by":"publisher","first-page":"260","DOI":"10.1053\/j.semdp.2019.07.002","article-title":"Phosphaturic mesenchymal tumors: a review and update","volume":"36","author":"Folpe","year":"2019","unstructured":"Folpe ALPhosphaturic mesenchymal tumors: a review and update. Seminars in Diagnostic Pathology201936260\u2013268. (https:\/\/doi.org\/10.1053\/j.semdp.2019.07.002)10.1053\/j.semdp.2019.07.002"},{"key":"bib3","series-title":"American Journal of Surgical Pathology","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1097\/00000478-200401000-00001","article-title":"Most osteomalacia-associated mesenchymal tumors are a single histopathologic entity: an analysis of 32 cases and a comprehensive review of the literature","volume":"28","author":"Folpe","year":"2004","unstructured":"Folpe ALFanburg-Smith JCBillings SDBisceglia MBertoni FCho JYEcons MJInwards CYJan de Beur SMMentzel TMost osteomalacia-associated mesenchymal tumors are a single histopathologic entity: an analysis of 32 cases and a comprehensive review of the literature. American Journal of Surgical Pathology2004281\u201330. (https:\/\/doi.org\/10.1097\/00000478-200401000-00001)10.1097\/00000478-200401000-00001"},{"key":"bib4","series-title":"American Journal of Surgical Pathology","doi-asserted-by":"publisher","first-page":"1371","DOI":"10.1097\/PAS.0000000000000890","article-title":"Phosphaturic mesenchymal tumors: clinicopathologic, immunohistochemical and molecular analysis of 22 cases expanding their morphologic and immunophenotypic spectrum","volume":"41","author":"Agaimy","year":"2017","unstructured":"Agaimy AMichal MChiosea SPetersson FHadravsky LKristiansen GHorch RESchmolders JHartmann AHaller FPhosphaturic mesenchymal tumors: clinicopathologic, immunohistochemical and molecular analysis of 22 cases expanding their morphologic and immunophenotypic spectrum. American Journal of Surgical Pathology2017411371\u20131380. (https:\/\/doi.org\/10.1097\/PAS.0000000000000890)10.1097\/PAS.0000000000000890"},{"key":"bib5","series-title":"BMC Musculoskeletal Disorders","article-title":"Treatment and outcomes of tumor-induced osteomalacia associated with phosphaturic mesenchymal tumors: retrospective review of 12 patients","volume":"18","author":"Zuo","year":"2017","unstructured":"Zuo QYWang HLi WNiu XHHuang YHChen JYou YHLiu BYCui AMDeng W. Treatment and outcomes of tumor-induced osteomalacia associated with phosphaturic mesenchymal tumors: retrospective review of 12 patients. BMC Musculoskeletal Disorders201718 403. (https:\/\/doi.org\/10.1186\/s12891-017-1756-1)"},{"key":"bib6","series-title":"Endocrine Journal","doi-asserted-by":"publisher","first-page":"675","DOI":"10.1507\/endocrj.EJ16-0587","article-title":"The diagnostic dilemma of tumor induced osteomalacia: a retrospective analysis of 144 cases","volume":"64","author":"Feng","year":"2017","unstructured":"Feng JJiang YWang OLi MXing XHuo LLi FYu WZhong DRJin JThe diagnostic dilemma of tumor induced osteomalacia: a retrospective analysis of 144 cases. Endocrine Journal201764675\u2013683. (https:\/\/doi.org\/10.1507\/endocrj.EJ16-0587)10.1507\/endocrj.EJ16-0587"},{"key":"bib7","series-title":"BMJ Case Reports","article-title":"Oncogenic osteomalacia due to phosphaturic mesenchymal tumour in the upper thoracic spine","volume":"13","author":"Garg","year":"2020","unstructured":"Garg BMehta NGoyal AKhadgawat R. Oncogenic osteomalacia due to phosphaturic mesenchymal tumour in the upper thoracic spine. BMJ Case Reports202013 e238209. (https:\/\/doi.org\/10.1136\/bcr-2020-238209)"},{"key":"bib8","series-title":"PLOS ONE","article-title":"Differential immune landscapes in appendicular versus axial skeleton","volume":"17","author":"Ahmed","year":"2022","unstructured":"Ahmed AAStrong MJZhou XRobinson TRocco SSiegel GWClines GAMoore BBKeller ETSzerlip NJ. Differential immune landscapes in appendicular versus axial skeleton. PLOS ONE202217 e0267642. (https:\/\/doi.org\/10.1371\/journal.pone.0267642)"},{"key":"bib9","series-title":"American Journal of Physiology-Endocrinology and Metabolism","doi-asserted-by":"publisher","first-page":"E254","DOI":"10.1152\/ajpendo.90201.2008","article-title":"Pathogenic role of FGF23 in Dmp1-null mice","volume":"295","author":"Liu","year":"2008","unstructured":"Liu SZhou JTang WMenard RFeng JQQuarles LD. Pathogenic role of FGF23 in Dmp1-null mice. American Journal of Physiology-Endocrinology and Metabolism2008295E254\u2013E261. (https:\/\/doi.org\/10.1152\/ajpendo.90201.2008)10.1152\/ajpendo.90201.2008"},{"key":"bib10","series-title":"Bone","article-title":"Challenges in the management of tumor-induced osteomalacia (TIO)","volume":"152","author":"Brandi","year":"2021","unstructured":"Brandi MLClunie GPRHouillier PJan de Beur SMMinisola SOheim RSeefried L. Challenges in the management of tumor-induced osteomalacia (TIO). Bone2021152 116064. (https:\/\/doi.org\/10.1016\/j.bone.2021.116064)"},{"key":"bib11","series-title":"Clinical Orthopaedics and Related Research","doi-asserted-by":"publisher","first-page":"3618","DOI":"10.1007\/s11999-013-3178-1","article-title":"The phosphaturic mesenchymal tumour: why is definitive diagnosis and curative surgery often delayed?","volume":"471","author":"Ledford","year":"2013","unstructured":"Ledford CKZelenski NACardona DMBrigman BEEward WC. The phosphaturic mesenchymal tumour: why is definitive diagnosis and curative surgery often delayed?Clinical Orthopaedics and Related Research20134713618\u20133625. (https:\/\/doi.org\/10.1007\/s11999-013-3178-1)10.1007\/s11999-013-3178-1"},{"key":"bib12","series-title":"Journal of Bone and Mineral Research","doi-asserted-by":"publisher","first-page":"469","DOI":"10.1002\/jbmr.3903","article-title":"Nonremission and recurrent tumor-induced osteomalacia: A retrospective study","volume":"35","author":"Li","year":"2020","unstructured":"Li XJiang YHuo LWu HLiu YJin JYu WLv WZhou LXia YNonremission and recurrent tumor-induced osteomalacia: A retrospective study. Journal of Bone and Mineral Research202035469\u2013477. (https:\/\/doi.org\/10.1002\/jbmr.3903)10.1002\/jbmr.3903"}],"container-title":["Endocrinology, Diabetes &amp; Metabolism Case Reports"],"original-title":[],"link":[{"URL":"https:\/\/edm.bioscientifica.com\/view\/journals\/edm\/2022\/1\/EDM22-0344.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/edm.bioscientifica.com\/downloadpdf\/journals\/edm\/2022\/1\/EDM22-0344.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/edm.bioscientifica.com\/downloadpdf\/journals\/edm\/2022\/1\/EDM22-0344.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T13:48:03Z","timestamp":1784555283000},"score":1,"resource":{"primary":{"URL":"http:\/\/journals.bioscientifica.com\/edm\/article\/3503"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,12,1]]},"references-count":12,"URL":"https:\/\/doi.org\/10.1530\/edm-22-0344","relation":{},"ISSN":["2052-0573"],"issn-type":[{"value":"2052-0573","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,12,1]]}}}